Advances in Management of Laryngeal and Subgiottic Stenosis

By Marshall St rome and Patricia K. Donahoe

Boston, Massachusetts

�9 The introduct ion of c ryotherapy and the ref ine- ment of surgical techniques has promoted progress in the care of infants and children w i th previously unresponsive intraluminal lesions of the larynx end subglott ic area. Three cases i l lustrate the plural ist ic approach that of ten must be brought to and individu- alized for these diff icult cases before a measure of success can be assured. One case, stenot ic since intubat ion required when the child was less than 1 k, was t reated w i th fulgurat ion, cryotherapy, and pro- longed stenting. The severe stenosis f rom the t ra- cheotomy site to the vocal cords eventual ly required open repair w i th anter ior and poster ior cricoid splits and a hyoid pedicled bone graft, fo l lowed by pro- longed stenting. A method of internal stent ing through the cords w i th percutaneous f ixat ion was effect ive for long-term (3-6 me) therapy. Another pat ient w i th severe stenosis was managed w i th mult iple cryotherapy procedures fo l lowed by pro- longed internal stenting. A th i rd pat ient w i th sub- glott ic stenosis secondary to an inhalat ion burn, was t reated w i th cryotherapy w i thou t stenting. This pre- sentat ion discusses the techniques of internal s tent- ing w i th percutaneous f ixat ion, cyrotherapy, and open repair w i th combined anter ior and poster ior cricoid split, considering the indications and idiosyn- cracies of each technique.

INDEX WORDS: Subglott ic stenosis; cryotherapy; an ter io r -pos ter io r cricoid split.

S UBGLOTTIC STENOSIS continues to be a most difficult problem, as attested to by the

number of modalities proposed for management, including medical therapy, dilatation, steroid injection, fulguration, cryotherapy, the laser, and open reconstruction. Choosing the appropriate treatment requires an understanding of the natu- ral evolution and the histopathologic staging of the injury, as well as a familiarity with all modalities that can be applied to its treatment. Three cases are discussed in which cryosurgery and/or open reconstructive surgery were used to mange this entity.

CASE REPORTS

Case I

This three-yr-old boy was transferred to the Boston Shrin- er's Burn Institute from Memphis, Tenn. 12 hr after sustain- ing severe flame burns involving 85% of the surface area. Multiple excisions and grafts were performed. Prolonged intubation compounded the inhalation injuries and necessi- tated a tracheotomy. Bronchoscopy was performed 3 mo later

and a fixed stenosis, 5 m m below the vocal cords was noted. Cryosurgery was performed 3 me later. The probe was applied to the stenosis, an anterior arc involving 60% of the circumference of the lumen, at - 50~ for 30 see. Side-biting forceps removed the frozen tissue during the short interval before hyperemia ensued) 'z Neither a stent nor a T- tube were inserted. The stenosis was inspected and dilated weekly for 2 wk, with steroids being injected on each occasion. Systemic Decadron was given for 4 wk. Epithelialization was complete in 2 wk, and extubation effected in 6 wk. Discussion. The applicability of cryosurgery as an isolated procedure in this case underscores the necessity for an under- standing of the physiologic considerations in injuries of the laryngotrachea. Three basic stages of injury have been described that can serve as a reference for management: (1) edema alone; (2A) infected mucosa as an isolated entity; (2B) infected mucosa with involvement of the internal perichon- drium, and (3) chondritiS with or without necrosis. 3 Medical management alone will almost uniformly suffice for stage 1. Stage 2A and 2B, if detected early, will also respond to medical management, which includes the use of steroids, appropriate antibiotics based on culture sensitivies of endo- scopicaUy removed tissue and judicious dilatation when neces- sary. The laser, although not an essential adjunct for early stage 2 pathology, can prove beneficial in the early infections stage by decreasing the volume of exuberant granulation tissue, thereby increasing the intralaminal dimension immedi- ately. Similarly, dilatation and cryosurgery can also increase iutraluminal dimensions in early stage 2. If undetected, this stage of injury progresses to soft tissue stenosis. This does not include cartilaginous contracture. Soft tissue stenosis if detected within 3 me of the initial injury often responds favorably to dilatation and concomitant intraluminal steroid injections. After 3 me, the efficacy of dilatation diminishes and we no longer recommed these more simple measures, when either cyrosurgery or laser surgery can be used successfully. If a short stenotic area produces clinical symptoms and a tracheotomy has not been performed, the laser is preferable in this setting to cryosurgery in that there is no associated swelling and tracheotomy could potentially be avoided. When a tracheotomy is present with soft tissue stenosis, either technique is applicable.

From the Division of Pediatric Surgery, Department of Surgery, Massachusetts General Hospital; Division of Oto- laryngology, Department of Surgery, Brigham & Women's Hospital, Beth Israel Hospital; and Harvard Medical School, Boston, Mass.

Presented before the 30th Annual Meeting of the Surgical Section of the American Academy of Pediatrics, New Orleans, La, October 31-November l, 1981.

Address reprint requests to Patricia K. Donahoe, M.D., Pediatric Surgery, Massachusetts General Hospital, Boston, MA 02114.

�9 1982 by Grune & Stratton, Inc. 0022-3468/82/1705~030501.00/0

Journal of Pediatric Surgery, Vol. 17, No. 5 (October), 1982 591

592 STROME AND DONAHOE

In this case, thickening had been present for 9 mo, a tracbeotomy was in place, and the stricture was moderately thick. Therefore, cryotherapy was chosen. Thirty seconds of application of the cryoprobe was adequate. Stenting was not required. Antibiotics, systemic steroids, dilatations and ste- roid injections were adjunctive measures used.

Case 2 K.M. was first seen at 11 yr of age with a history of

recurrent severe asthma attacks, recurrent laryngospasm, and cardiorespiratory arrests requiring multiple endotracheal intubations. An anterior myocardial infarct, episodes of pulmonary edema, neurologic sequelae, including a severe seizure disorder, and a severe depressive reaction complicated these events. A trache0t0mY was performed because of an increasing frequency o f laryngospasm and the prolonged intubations these episodes required. Bronchoscopy initially showed fibrinous exudates over the false cords, and adjacent 1 em of the subglottic area. One month later, the area described was found to have contracted with a noted decrease in intraluminal dimension. Dilatations at two successive monthly intervals did not have a positive effect.

Four months later, a cryotherapy probe (Frigitronics Cor- poration, Shelton, Conn.), courtesy of Dr. Bradley Rogers, was applied for 35 sec at - 8 0 ~ from 12 o'clock to 7 o'clock on the right. Frozen tissue was removed with cup forceps before hyperemia ensued. A stent was placed from the tracheotomy site to the false vocal cords. The stent was removed after 1 too. After 2 mo, re-epithelialization had occurred and the tracheotomy was removed. Stridor at rest and dyspnea slowly recurred. T-tube steuting was attempted 3 mo after cryosurgery, but the tube occluded after 5 days and was removed. The tracheotomy was re-inserted. Six months after cryosurgery and stenting, an open surgical approach was contemplated. Pre-operative endoscopy revealed a satisfactory tracheal lumen and a posterior glottic stenosis. The latter was incised endoscopically to cartilage and injected with steroids on both sides. Subsequently, she was extubated and remains so. Her voice has a coarse quality. Discussion. This case is representative of a long Iongititudi- nal stenusis with cartilage involvement responsive to cryother- apy. The reported degree of injury represents stage 3 in the developmental spectra. When cartilage becomes infected, secondary eontracture frequently develops and some degree of stenosis ensues. Internal perichondrium and cartilage infec- tion, if treated aggressively medically within 1 wk of the onset of pathology, will often respond favorably with a resultant adequate airway, as long as necrosis has not lead to sequestra- tion. Beyond I wk, the incidence of stenosis requiring surgical management increases dramatically. With cartilage contrac- ture and subsequent fibrosis, the laser has been far less effective than with soft tissue injury alone. It is here, however, that the unique aspects of cryosurgery are especially applica- ble. 4 In 1971, one of us (M.S.) reported on the effects of cryosurgery in the canine larynx and the histopathologic stages of the cryosurgically-induced laryngeal lesion, s Injury to cartilage can he induced with cryosurgery, but neovascular- ization is evident with subsequent healing if external perichon- drium is preserved. In this patient, scar, internal perichon- drium, and cartilage were frozen with a 45-sec freeze. On thawing, a predominantly rigid area softened and an intralu-

menal stent, properly measured, provided sufficient support while cartilaginous healing was effected, giving stability with increased dimension. We would now recommend a longer initial interval of internal stenting.

Technique of Endoscopic Freezing The involved areas are exposed endoseopically with the

Dedo laryngoscope for subglottic lesions and the bronchus- cope for tracheal lesions. We modified the Frigitronics endo- laryngeal probe developed by Rogers, t'2 by shortening the length of the freezing area to 3 ram. The conductive area of the probe is juxtaposed to the stenotic area for the freeze interval, which is based upon the patient's age, the luminal size, circumference of the stenosis, and the magnitude of scarring. For children under 3 yr of age, a 30-sec freeze interval is all that is warranted as an initial procedure. For teens and adults, the experienced operator may choose an expanded freeze interval, not to exceed 1 rain. It is essential to err on a shorter, rather than a longer freeze interval. Too long a freeze can lead to destruction of the external perichon- drium, and negate the potential for phyisologic repair, thus making reconstructive efforts--with the possible exception of tracheal resection---extremely difficult, if not impossible.

In children under 3 yr of age with significant stenoses, the cryoprobe may fill the stenotic area. In this situation, the probe can be accurately placed if the stenotic area is measured both endoscopically and radiographically. A tight fitting probed leads to a circumferential freeze. Today, we no longer remove the frozen tissue with a forcep, as removal can cause unnecessary contracture. 4 Delayed spontaneous sepa- ratiou of the treated area will occur. Histopathologic section- ing of frozen tracheal segments has shown that a 30-sec freeze in the young child will cause necrosis of internal perichondrium and limited adjacent cartilage (Strome and Donahoe, unpublished data). Therefore, with a circumferen- tial freeze, stenting is mandatory and has the potential for increasing luminal dimensions.

We recommend the largest possible stent that will pass. Portex anesthesia tubing serves nicely and is treated as originally described by Birk. 6 An anterior longtitudinal or horizontal keel is created for placement at the level of the true vocal cords and the stent endoscopically positioned and fixed as follows (Figs. 1, 2).

Double 2-0 proline is passed through the trachea on a Keith needle while the carotid is protected with the opposite hand. Darkening the operating room, with a telescope or bronchoscope in the trachea, will facilitate external identifi- cation of the correct tracheal level for suture passage in difficult scarred cases. Suture material is then brought out through the bronchoscope into the mouth, and affixed to an appropriately measured and heat-treated stent. The stent is placed into the subglottic site and secured by tying the suture externally over a button.

Antibiotics are used for 1 wk post stent placement, and thereafter, only if the patient contracts an upper respiratory tract infection. Decadron is given at the time of endoscopy and for 48 hr.

The patient is endoscoped at 3 mo and the duration of stenting determined by the amount of collapse observed. If significant collapse persists, then a new stent is re-introduced for an additional 3 mo interval.

At the time of stent removal, all children will have varying

|

Direct vision

Needle in side of neck

f ~ I =:=

B

�9 True cord

|

| Grasp suture, withdraw through moutl

) i~ / /

Divide end of loop; G pass ends through

stent and tie

| Stent placed through laryngoscope

gJ~

tied over button ~.. ~~~"=~

Crimp of stent at level of cord

Fig. 1. (A) Transluminal stents are placed by passing a straight needle percutaneously anter ior to the sternocleidomas- told, through the trachea under bronchoscopic guidance. (B) The needle delivers a 2-0 prol ine suture through the tracheal lumen where it is grasped by forceps passed down the bronchoscope. The eye of the needle is broken to release the suture. (C) 2-0 prol ine suture is drawn up the bronchoscope and out the mouth, The suture is divided and passed through a careful ly measured stent, which has been keeled to pro tec t the vocal cords. The keel is created by loosely applying a straight snap a|ong the end or across a Por tex endotracheal tube, which is then flash steri l ized (Fig. 3). (D) The keeled tube is careful ly measured to f i t the endoscopically and radiographical ly measured distance f rom the t racheotomy tube to a point above the vocal cords. The suture is t ied outside of the pierced stent, which is posit ioned via the bronchoscope. The suture is then securely t ied over an external but ton.

593

594 STROME AND DONAHOE

Fig. 2. The keel s tont is made by flash sterilizing n straight snap on a Por tex tube cut to fit the child's stenot ic defect, The keel can be made either longitudinal, as in (A) , or horizontal, as in (B).

degrees of granulation tissue as a reaction to the stent. This is not a eontraindication to removal, which is predicated solely upon the degree of collapse and the intraluminal dimensions as determined by the ability to easily pass an endoscope of appropriate size. Resolution of the granulation tissue may take several months. Patience is required. Steroids appear to be beneficial at this stage and we use them in conjunction with antibiotics until mucosalization is complete. The tra- cheotomy tube is removed when mucosalization, as deter- mined by endoscopy, is complete and the lumen judged adequate for the child's given age.

Currently, we are using cryosurgery in moderate to severe stenoses, when a tracheotomy is present, and cartilaginous contracture suggests laser management may be less than optimal. If the cryosurgically-induced lesion allows the intro- duction of the stent that maintains normal tracheal dimen- sions for the patient's age, the outlook for success with this therapeutic approach is optimistic. Conversely, if after cryo- destruction, an appropriately sized stent cannot be placed, an open procedure at a later date will be necessary. Similarly, an open procedure is indicated for cryosurgical failure.

Open repair can obviously be used as an initial procedure for severe stenosis. However, if cryosurgery is used as described, the necessity for such will be reduced.

Case 3

A premature infant weighing 1 kg required endotracheal intubation at birth because of respiratory distress. Adequate ventilation could not be maintained with a 2.5 Portex endo- tracheal tube, so a slightly tight, 3.0 Portex tube had to be

inserted to insure necessary ventilation, At the time of a tube change, bronchoscopy revealed edema and some erythema with a patent airway, and the 3.0 Portex tube was replaced. After 3 mo of intubation, a tracheotomy was performed. One month later, bronchoscopy revealed a thick, elongated stric- ture in the subglottic area with only a 2-mm opening. The true vocal cords were normal. Four months later, at 8 mo of age, cryotherapy was performed for 30 sec at - 8 0 ~ at the narrowest stricture site. The freeze was circumferential; therefore, we fashioned a 2.5 Portex stent, which was placed endoscopically and fixed percutaneously. Cryotherapy for 45 sec at - 80~ was repeated in 2 mo on the left side, and the frozen tissue removed from that half with side-biting forceps. A 3.5 stent was introduced and the tracheotomy site repaired.

Three months later, the stent was removed. The true vocal cords looked normal. Steroids and antibiotics were instituted. Within 2 mo, the cryotherapy was repeated on the right side of the stenosis, and a 3.0 stent reinserted. The stented area was inspected at 1 and 3 rap, with placement of 3.5 and 4.0 stents. Because the vocal cords were slightly swollen after 4 rap, with the transglottic stent in place, and the trachea was collapsing at the traeheotomy site, a T-tube was inserted from the tracheotomy to the subglottic space.

Over a 3-mo period, despite multiple alterations in the length of the T-tube, recurrent pneumonia and increasing subglottic and glottic granulations necessitated its removal. Subsequently, the distal subglottic area slowly epithelialized, while the more proximal area became very stenotic, despite multiple steroid injections. We then elected to do an open repair.

Anterior and posterior cricoid split with a vascularized hyoid bone graft was performed and the region stented with a 5.5 Portex endotracheal tube (Fig. 3). That portion of the bone graft not attached to muscle was covered with a pediele of sternohyoid muscle distally. The hyoid graft remained in good position and the stent had produced no significant reaction, although left in position for 6 mo when it was removed. The tracheotomy was replaced with progressively small tubes and was intermittently plugged to encourage vocalization. Speech therapy was instituted. The tracheot- omy tube was removed 18 mo after open repair. The left cord moved normally, and closed against the partially mobile right hemilarynx. The child is learning to verbalize, speaking with a deep, coarse vocal quality. Discussion. Tracheotomy should be considered earlier in the premature infant, rather than submitting them to pro- longed intubation, if an endotracheal tube larger than ideal is necessary to maintain ventilation. We would now consider an early anterior cricoid split as an alternative. 7 In this age group, cryotherapy should he performed for 30 sec or less at - 8 0 ~ One can stent through the vocal cords for greater than 6 mo without significant ill effect. T-tube maintenance was extremely difficult in this child. The excursions that motion of the head produce with one end of the tube butressed at the traeheotomy site produced trauma to the subglottic area and vocal cords, inducing stenosis. Simultaneous anterior and posterior cricoid split can be performed successfully, but require prolonged internal stenting, s

Before undertaking open surgical repair, mucosalization must he complete over the underlying cicatrix in order to achieve the best possible results. Our current operative tech-

LARYNGEAL AND SUBGLO'I-rlC STENOSIS

Q Mobilize hyoid bone |

Thyroid cartilage and trachea

divided in midline

595

Post Iomil cric~ divk

\

Trocheostomy

%

~Thyroid carl.

"~'Cricoid

Scar tissue

d r . . . . . . . J i ~ L

|

Stint in

lumen

Intact Hyoid pedicle bone graft in place

Muscle to cover hyoid bone graft

Fig. 3. (A) Case 3. The ex ten t of stenosis is depicted, involving the subglott ic space and trachea. (B) An anter ior cricoid and midline tracheal spli t has been performed. The intraluminal space was not appreciably increased, Mucosal ized scar was not removed. (C) The poster ior cricoid has been split and spread, increasing intraluminal dimensions, The pedicled hyoid graf t has been defined. (0) A large stent is placed and secured. The hyoid graf t is t ransposed into the newly created anter io r space.

596 STROME AND DONAHOE

nique leaves untouched the mucosalized scar, preferring to avoid earlier methods of removal of all scar tissue and muensa with resurfacing, using either mueosa or skin grafts. The entire longitudinal area of stenosis is opened via a midlioe approach. A stair-step midline incision is gaining popularity. 9 if sufficient space is achieved by a midline incision alone, the stent is placed maintaining tbls new dimension, and an ante- rior graft is inserted. Costal cartilage, free hyoid and vascular- ized hyoid have been used successfully. In selected instances, with only an anterior split, a very stable graft may be left unstented.

When an anterior split does not provide adequate intralumi- nal dimension, increased space can he gained by splitting the posterior cricoid lamina, l~ The interaryteuoideus muscle should also be divided, as this maneuver improves the postop- erative vocal quality, s We do not recommend free grafts to maintain the newly created posterior space. Experience in animals has shown that these do not survive (1) because of significant motion in that area following the posterior split, and (2) because grafts can serve as a potential nidus for infection. Thus, the posterior lamina is distracted as far as possible and the largest permissible intraluminal stent secured. The stent is fashioned and fixed as previously described. The pedicled, vascularized, byoid graft is secured in the newly

developed anterior glottic and tracheal space. It is our percep- tion that the posterior split affords greater space anteriorly by allowing distraction of the laryngeal halves. In this case, no increased dimension was achieved initially with the anterior split alone, but a concomitant posterior split provided a good lumen. Intraluminal stents should be left in position for longer than 3 too. The anterior cutaneous scar can serve as a useful clinical clue for stent removal. After 3 mo, if the scar has softened and there is no evidence of collapse intraluminally, the stent can usually he removed quite successfully. If the scar is still firm, the stent should remain in place longer.

SUMMARY

T h e i n d i c a t i o n s a n d l i m i t a t i o n s o f c r y o s u r g e r y

as wel l as a d j u n c t s t e n t i n g w e r e d i s cus sed . C r y o -

s u r g e r y , i f u sed j u d i c i o u s l y , h a s a p l a c e in t h e

l a r g e r a r m a m e n t a r i u m of m o d a l i t i e s t h a t c a n be

a p p l i e d to p a t i e n t s w i t h s u b g l o t t i c s tenos i s . T h e

r a t i o n a l e for a n t e r i o r - p o s t e r i o r s p l i t t i n g o f t h e

c r i co id a n d o u r p r e f e r e n c e fo r a d j u n c t g r a f t i n g

a n d s t e n t i n g w e r e d e t a i l e d ,

REFERENCES

1. Rogers BM, Rosenfeld M, Talbert JL: Endobronehial cryotherapy in the treatment of tracheal strictures. J Pediatr Surg 12:443-449, 1977

2. Rogers BM, Talbert JL: Clinical applications of endo- tracheal cryotherapy. J Pediatr Surg 13:662-668, 1978

3. Strome M, Ferguson CF: Multiple post intubation complications. Ann Oto[ Rhinol Laryngol 83:432-438, 1974

4. Li AKC, Ehlich HP, Trelstrad RL, et al: Differences in healing of skin wounds caused by burn and freeze injuries. Ann Surg 191:244-248, 1980

5. Strome M: Cyrosurgery: The effect on canine endola- ryngeal structures. Laryngoscope 81: 1057-1065, 1971

6. Birck HG: Endoscopic repair of laryngeal stenosis. Am Acad Ophthalmol Otol 74:140-143, 1970

7. Cotton RT, Seid AB: Management of the extubation problem in the premature child. Anterior cricoid split as an alternative to tracheotomy. Ann Otol Rhinol Laryngol 89:508-511, 1980

8. Strome M: An assessment of autogenous grafts in the posterior cricoid lamina (in press)

9. Evans JNG, Todd GB: Laryngotracheoplasty. J Laryn- gol 88:589-597, 1974

10. Crysdale NS, Platt L J: Division of the posterior cricopharyngeal plate in young children with subglottic ste- nosis. Laryngoscope 86:1451-1458, 1976

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